Sarcoidosis Presenting Solely As Hiccups

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An Uncommon Cause of Hiccups: Sarcoidosis Presenting Solely as Hiccups

Lien-Fu Lin*, Pi-Teh Huang Division of Gastroenterology, Department of Internal Medicine, Tung’s Taichung Metroharbor Hospital, Taichung, Taiwan, R.O.C.

Common causes of hiccups are over-distension of the stomach, a sudden change in gastrointestinal temperature, exces- sive alcohol and tobacco ingestion, and sudden excitement or emotional changes. Common presenting symptoms of sarcoidosis include cough, dyspnea, and chest pain. It is very rare for a sarcoidosis patient to present with hiccups. A 48-year-old man presented with hiccups of 2 weeks’ duration. He denied having headaches, earache, cough, abdominal pain, fever, or body weight loss history. On physical examination, he had no peripheral lymphadenopathies in the neck, axilla and inguinal regions, no organomegaly in the abdomen and no skin abnormalities. A neurological examination showed normal findings. Laboratory investigations revealed a normal complete blood count, liver function, renal function, serum calcium, and tumor markers. Transabdominal ultrasound was negative, and panendoscopy revealed a small healing duodenal ulcer. Chest radiography showed an enlarged right lung hilum, while computed tomography showed enlargement of multiple mediastinal lymph nodes. Endoscopic ultrasound-guided fine-needle aspiration with a 22-gauge needle and trucut biopsy with a 19-gauge needle (quick-core biopsy needle) were performed, and cytology, cell block and histology revealed non-caseating granuloma, with negative tuberculous and fungus cultures. Mediastinal lymph node due to sarcoidosis can be a rare cause of hiccups. [J Chin Med Assoc 2010;73(12):647–650]

Key Words: endoscopic ultrasound-guided fine-needle aspiration, hiccups, mediastinal lymph node, sarcoidosis

Introduction abdominal pain, fever, or body weight loss. On physical examination, he had no peripheral lymphadenopathies A hiccup is an involuntary contraction of the diaphragm in the neck, axilla and inguinal regions, no organo- and intercostal muscles, causing sudden inspiration megaly in the abdomen, and no skin abnormalities. and abrupt closure of the glottis. The reflex arc of a Neurological examination results were normal. hiccup is composed of an afferent limb, including the Laboratory investigations revealed a normal com- vagus and phrenic nerves, sympathetic chain, a central plete blood count, liver function, renal function, serum mediator, and an efferent limb, including the phrenic calcium, globulin and tumor markers. Transabdominal nerve, with accessory efferent neural connections to the ultrasound was negative, and panendoscopy showed a glottis and inspiratory intercostal muscles.1 Common small healing duodenal ulcer. Chest radiography re- causes of hiccups are over-distension of the stomach, vealed an enlarged right lung hilum (Figure 1), while a sudden change in gastrointestinal temperature, exces- computed tomography showed multiple mediastinal sive alcohol and tobacco ingestion, and sudden excite- lymph node enlargement (Figure 2). Endoscopic ment or emotional changes.2 We present a case of ultrasound-guided fine-needle aspiration (EUS-FNA) sarcoidosis who presented solely with hiccups. with a 22-gauge needle (Echotip; Cook Endoscopy, Winston-Salem, NC, USA), and trucut biopsy (quick- core biopsy needle; Cook Endoscopy) were performed Case Report (Figure 3). Cytology, cell block and histology (Figure 4) showed non-caseating granuloma, with no acid-fast A 48-year-old man presented with hiccups of 2 weeks’ bacilli, and cultures for Mycobacterium tuberculosis duration. He denied having headaches, earache, cough, and fungus were negative. Polymerase chain reaction

*Correspondence to: Dr Lien-Fu Lin, 8, 11th Floor, Mei Tsun Road, Section 2, Lane 168, Taichung 402, Taiwan, R.O.C. E-mail: [email protected] ● Received: November 30, 2009 ● Accepted: May 7, 2010

Figure 1. Enlarged right hilar region (arrow).

R L 1 1 7 7 5 5 Figure 3. (A) Endoscopic ultrasound (EUS)-guided fine-needle aspiration with a 22-gauge needle (arrow) from mediastinal lymph nodes. (B) EUS-guided trucut biopsy with a 19-gauge needle (arrow) from a mediastinal lymph node.

B of sarcoidosis. Lung function tests (lung volume and diffusion capacity) were normal. There were no lesions in the eyes after consultation with an ophthalmologist. The patient was given steroids, but could not tolerate the steroid therapy. He was kept under observation and followed-up for 4 months, but still had a hiccup R L 1 1 frequency of 4–5 times a day. 7 7 5 5

Discussion

The reflex arc of a hiccup consists of an afferent pathway, including the vagus and phrenic nerves, sympa- Figure 2. (A, B) Multiple enlarged mediastinal lymph nodes can be seen on chest computed tomography (arrows). thetic chain, a central mediator, and an efferent limb, including the phrenic nerve.1 Any lesions involving this pathway can cause hiccups. There were no central for M. tuberculosis was negative. Serum angiotensin- nervous system, ear, throat, neck, metabolic and intra- converting enzyme level was elevated to 27.2 IU/L abdominal causes in our patient to explain the etiology (normal, 8.3–21.4 IU/L). of hiccups, except for the finding of mediastinal lymph The findings of mediastinal lymph node enlargement, nodes. non-caseating granuloma, exclusion of tuberculous Lymphoma, mediastinal cysts, and neurogenic neo- and fungal infections, and an elevated angiotensin- plasms are the most common primary middle and converting enzyme level led to the clinical diagnosis posterior mediastinal tumors.3 Other lesions include

648 J Chin Med Assoc • December 2010 • Vol 73 • No 12 Sarcoidosis presenting solely as hiccups

was due to compression of the phrenic nerve in the A mediastinum. Diagnosis of sarcoidosis is dependent on compati- ble clinical and radiographic features, accompanying histological evidence of non-caseating epithelioid-cell granulomas, for which there are no apparent causative organisms or particles.7,8 Tissue diagnosis can be ob- tained from the mediastinum via EUS-FNA, rather than by mediastinoscopy. EUS-FNA can obtain gran- ulomatous material in sarcoidosis with a high degree of accuracy,9–11 and 1 retrospective study has found the sensitivity to be 89% with a specificity of 96% for EUS-FNA for diagnosing granuloma in sarcoidosis.12 Mediastinoscopy is also more invasive than EUS-FNA, B and can have a morbidity of 16%, with even 1 mortality occurring.13 EUS-FNA is very safe, with a complication rate of only 0.8%, as reported in a meta-analysis.14 Furthermore, EUS-guided trucut biopsy can be helpful in cases missed by EUS-FNA.15 Most patients with sarcoidosis do not require therapy. Treatment for pulmonary sarcoidosis is best guided by pulmonary-function studies.8 Two-thirds of patients with sarcoidosis usually have remission within a decade after diagnosis, with few consequences; more than half of patients have remission within 3 years.8 Fewer than 5% of patients die from sarcoidosis, and death is usually the result of pulmonary fibrosis with respiratory 8 C failure or cardiac or neurological involvement. In conclusion, gastroenterologists should be aware that mediastinal lymph node due to sarcoidosis could be a cause of hiccups.

References

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